Speech, Language and Communicative Ability in School-Aged Children with Cerebral Palsy and Speech
Impairment
Ann Nordberg
Division of Speech and Language Pathology Institute of Neuroscience and Physiology Sahlgrenska Academy at University of Gothenburg
Gothenburg 2015
Cover photo by the author: Anemone hepatica, typical spring flowers of the mountain of Billingen, near the city of Skövde, Västergötland, Sweden, birthplace of the author.
Speech, Language and Communicative Ability in School-Aged Children with Cerebral Palsy and Speech Impairment
© Ann Nordberg 2015 ann.nordberg@neuro.gu.se ISBN 978-91-628-9503-7
Printed in Gothenburg, Sweden 2015 Ineko
To Björn and Erik, My father Hans, and in memory of my mother Maja
Speech, Language and Communicative Ability in School-Aged Children with Cerebral Palsy and Speech Impairment
Ann Nordberg
Division of Speech and Language Pathology, Institute of Neuroscience and Physiology, Sahlgrenska Academy at University of Gothenburg, Sweden
Abstract: The overall aim of this thesis was to explore and describe speech, language and communicative ability in school-aged children with cerebral palsy (CP) and speech impairment. The medical records of a population- based cohort of 129 children with CP born 1999–2002 in western Sweden were reviewed. Type of CP, motor functions and neuroimaging findings were analysed.
Twenty-seven (21%) of the 129 children had speech impairment. Twenty-two (82%) of the 27 children took part in an assessment of speech, language, and communication skills.
Oral consonant production, dysarthria, hypernasality and narratives were assessed. The children’s, the parents’, the teachers’ and the SLP’s opinions about the children’s communicative ability were also analysed.
More than half of the children had severe problems with articulation of oral consonants. Speech production and non-verbal cognitive level correlated significantly and severe retelling ability problems occurred. Language ability and auditory memory correlated significantly with retelling ability. The children were mostly positive about their own communication. Parents and teachers rated them with marked general communicative impairments. The parents’ and SLP’s ratings correlated significantly, whereas the parents and the teachers ratings did not.
A comprehensive speech, language and communication test battery including standardised tests is suggested. The children’s own opinions and those of key persons in their environment are also important to consider when planning intervention.
Keywords: cerebral palsy, children, epidemiology, neuroimaging, narrative ability, communicative ability, dysarthria, consonant articulation
ISBN: 978-91-628-9504-7
SAMMANFATTNING PÅ SVENSKA
Cerebral pares (CP) är den vanligaste motoriska funktionsnedsättningen hos barn och orsakas av en tidig hjärnskada. Förutom varierande motoriska problem kan barn med CP ha olika grader av svårigheter med exempelvis kognition, syn och kommunikation. Barnens verbala och icke-verbala kommunikationsförmåga kan vara negativt påverkad. Nedsatt funktion kan förekomma gällande andning, fonation, velofarynx och artikulation.
Kunskapen om tal, språk och kommunikation hos talande barn med CP är begränsad. Det övergripande syfte i denna avhandling var därför att undersöka detta hos skolbarn med CP och talstörning.
I Studie I klassificerades en populationsbaserad grupp av barn med CP i Västra Götaland födda 1999-2002 (n=129) genom journalgenomgång i kategorierna: 1) Utan talstörning 2) Talstörning 3) Icke-talande. Information om typ av CP, motorisk förmåga, kognitiv nivå och hjärnskadelokalisation fanns sedan tidigare. Resultatet visade att mer än hälften (53%) av den populationsbaserade gruppen av barn med CP saknade tal eller hade talstörning. Av de 27 barnen (21%) med talstörning undersöktes 22 (medelålder 11 år 3 mån) gällande tal-språk- och kommunikationsförmåga. I Studie II undersöktes talproduktion och icke–verbal kognitiv förmåga hos 19 (86%) av de 22 barnen. Grad av dysartri, hypernasalitet, procent korrekta orala konsonanter samt typ av artikulatoriska avvikelser analyserades.
Majoriteteten (84 %) bedömdes ha åtminstone lätt dysartri, men bara några få hade hypernasalitet. Över hälften hade däremot stora eller mycket stora problem med konsonantproduktionen . I Studie III undersöktes muntlig återberättarförmåga och andra språkliga och kognitiva förmågor hos 15 (68
%) av de 22 barnen. Sambandsanalyser och fördjupad analys av barnens återberättarförmåga utfördes. Grava svårigheter med återberättarförmåga, receptiv och expressiv språkförmåga, minne, icke-verbal kognition och
"theory of mind" förekom hos barnen som undersöktes. I Studie IV skattade barnen sitt eget tal och kommunikation, och barnens föräldrar, lärare och en logoped gjorde också skattningar av barnens kommunikation. Hälften av de 16 barn som deltog i studie IV var i stort sett positiva till sin kommunikation.
Barnens skattningar samvarierade inte signifikant med hur deras föräldrar och lärare skattade dem. Föräldrar och lärare skattade att mer än hälften av barnen hade grava kommunikativa svårigheter, men även deras skattningar skilde sig åt. Däremot skattade föräldrar och logopeden barnens kommunikativa förmåga på samma sätt.
Sammanfattningsvis visar denna avhandling att mer än hälften av barnen med CP ur den populationsbaserade gruppen hade talstörning eller saknade tal.
saknade tal. Hos barnen med talstörning var omfattande generella språkliga svårigheter vanligt liksom nedsatt icke-verbal kognitiv nivå. Föräldrar och lärare noterade att de flesta av barnen hade generella kommunikativa svårigheter och att mer än hälften hade grava svårigheter trots att en majoritet av barnen själva inte tyckte att de hade några problem.
Barn med CP och talstörning behöver uppmärksammas mer. Logopeder bör göra en noggrann bedömning av talproduktion kombinerad med bedömning av olika språkliga domäner inför ställningstagande till behandling. Hänsyn behöver också tas till barnens egna och omgivningens åsikter angående deras kommunikation.
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LIST OF PAPERS
This thesis is based on the following studies, referred to in the text by their Roman numerals.
I. Nordberg A., Miniscalco C., Lohmander A., &
Himmelmann, K. (2013). Speech problems affect more than one in two children with cerebral palsy: Swedish population- based study. Acta Paediatrica,102,161-166.
II. Nordberg A., Miniscalco C., & Lohmander A. (2014).
Consonant production and overall speech characteristics in school-aged children with cerebral palsy and speech impairment. International Journal of Speech-Language Pathology, 16, 386-395 (Invited Article, Special Issue Childhood Dysarthria).
III. Nordberg A., Dahlgren Sandberg A., & Miniscalco C.
(2015). Story retelling ability and language ability in school- aged children with cerebral palsy and speech impairment.
International Journal of Language & Communication Disorders. Published online: 27 July 2015.
IV. Nordberg A., Miniscalco C., Lohmander A., &
Himmelmann, K. (2015). Perspectives of communicative ability in children with cerebral palsy and speech
impairment. (Submitted).
Paper I is reprinted with kind permission from ©Foundation Acta Paediatrica 2013.
Paper II is reprinted with kind permission from International Journal of Speech-Language Pathology.
Paper III is reprinted with kind permission from International Journal of Language & Communication Disorders.
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TABLE OF CONTENTS
ABBREVIATIONS ... IV
1 INTRODUCTION ... 1
1.1 Cerebral Palsy ... 1
1.2 Clinical Management of Children with CP ... 1
1.3 International Classification of Functioning, Disability and Health (ICF) ... 3
1.3.1 Neuroanatomical correlates to speech and language impairment ... 4
1.3.2 Speech impairment ... 5
1.3.3 Language impairment ... 6
1.3.4 Speech and language assessment ... 8
1.3.5 Communicative activity and participation assessment ... 10
1.3.6 Own and environmental opinions about communicative ability ... 11
1.4 Rationale ... 13
2 AIM ... 14
3 MATERIALS AND METHODS ... 15
3.1 Participants ... 15
3.2 Ethical consideration ... 18
3.3 Procedures ... 18
3.3.1 Test instruments ... 18
3.3.2 Analyses ... 23
3.3.3 Statistical analyses ... 24
3.3.4 Reliability ... 26
4 RESULTS ... 28
4.1 Speech ability in relation to type of CP, motor function and neuroimaging findings (Study I) ... 28
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4.2 Speech characteristics (Study II) ... 30
4.3 The impact of language abilities on retelling ability (Study III) ... 30
4.4 Perspectives on the children’s communicative ability (Study IV) ... 31
5 DISCUSSION ... 32
5.1 Speech ability in relation to type of CP, gross motor function and neuroimaging findings ... 32
5.2 Speech characteristics ... 33
5.3 The impact of language abilities on retelling ability ... 35
5.4 Perspectives on the children’s communicative ability ... 36
5.5 Methods used in the studies ... 38
6 LIMITATIONS AND FUTURE RESEARCH ... 39
7 CLINICAL IMPLICATIONS ... 41
8 CONCLUSIONS ... 42
ACKNOWLEDGEMENT ... 44
REFERENCES ... 48
ABBREVIATIONS
CP Cerebral Palsy
CFCS Communication Function Classification System GMFCS Gross Motor Function System
ICF International Classification of Functioning, Disability and Health
ICF-CY International Classification of Functioning, Disability and Health for Children and Youth
SLP Speech and Language Pathologist BSCP Bilateral Spastic Cerebral Palsy USCP Unilateral Spastic Cerebral Palsy VSS Viking Speech Scale
WHO World Health Organisation
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1 INTRODUCTION
1.1 Cerebral Palsy
Cerebral palsy (CP) is the most common type of motor impairment in children, affecting about two children per 1000 live births (Stanley &
Watson, 1992; Himmelmann, Hagberg, & Uvebrant, 2010). CP is an umbrella term and is defined as a persistent impairment of motor function caused by non-progressive pathological processes in the immature brain (Surveillance of CP in Europe; SCPE, 2000). CP is classified into three main groups: spastic, dyskinetic and ataxic CP (SCPE, 2000). The time and reason the child receives the diagnosis of CP can vary, but the recommended age to determine the type of CP is 4–5 years. The motor impairment of CP is often accompanied by disturbances of sensation, perception, cognition, communication and behaviour, as well as epilepsy (Rosenbaum et al., 2007).
The epidemiology of CP and its accompanying impairments has been studied in the health care region of western Sweden since 1954. (Hagberg, Hagberg,
& Olow, 1975). This is one of the longest running studies of CP in the world.
In the last decade, also the communication of children with CP has been added to the descriptions of the accompanying impairments. Communication and speech ability has been described in children with dyskinetic CP born in the 1990s (Himmelmann, Hagberg, Wiklund, Eek, & Uvebrant, 2007), and in all children with CP in western Sweden since the birth year 1999 (Himmelmann & Uvebrant, 2011). Communicative ability has also been studied in a group of adolescents born 1991–1997 (Himmelmann, Lindh, &
Hidecker, 2013).
1.2 Clinical management of children with CP
Children with CP may have various communication impairments and experience difficulties with communicative ability in areas such as speech and language production, comprehension of language, gestures and facial expressions (Pennington et al., 2005). It has been reported that children with these impairments experience more limited participation in their daily lives than children with CP without communicative difficulties (Dickinson et al., 2007). There are currently no agreed standard assessment practices for children with CP and communication impairment in Sweden. However, there are recommendations made in the national care programme for children with CP (regional care programme cerebral palsy, 2014), a programme intended to
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serve as a knowledge base and support for healthcare professionals in the practical everyday work for the development of the quality of care of children with CP. The National Quality Register for Habilitation is a Swedish quality register for all children (0–18 years) with disabilities (HabQ; Öhrvall, Eliasson, Löwing, Ödman, & Krumlinde-Sundholm, 2010). It is a systematic register for developing and securing the quality and results of the activities that take place at the habilitation centres in parts of Sweden. Speech, language, eating abilities, dysphagia and communicative effectiveness, i.e.
total communicative ability including augmentative and alternative communication (AAC), are surveyed by the speech and language pathologists (SLPs) using the Therapy Outcome Measure (TOM; Enderby, 2014;
Enderby, John, & Petherham, 2006). Assessments with TOM are performed at 3, 6 and 12 years of age (and when possible at 15 years).
Communication impairments of children with CP are sometimes identified by the general speech, language and communication screening at 2.5–3 and/or at 4 years of age at the general child care health centre. The children with CP and their families are offered care at the habilitation centres. Various health professionals, such as speech language pathologists (SLPs), psychologists, physicians, occupational therapists, education specialists and physiotherapists at the centres work closely together in teams in order to meet the special needs of the child and her/his family. For children with major difficulties, a thorough assessment of speech, language, communication, cognition and oral motor function is performed at about age 3. Hearing and visual skills ought to be checked. The assessment may need to be repeated during the child’s language and communication development. Before starting school, additional information about reading and writing aids are provided, and when needed a new assessment of cognitive level is conducted.
Children with CP and severe communicative impairments can benefit from the provision of AAC systems, which can supplement existing speech or replace speech that is not functional. AAC methods vary and include for example signing, gestures, picture charts, books and special computers.
However, children with CP and speeech impairment, communicating with spoken language, seem to be heavily underserved according to the results of a survey to SLPs at the Health Habilitation Services at Stockholm County Council (Egefors, 2012). In this survey children with CP and speech impairment were not even identified at the habilitation centres and accordingly, neither assessed, nor received any speech intervention This group is therefore the focus of this thesis.
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1.3 International Classification of
Functioning, Disability and Health (ICF)
The International Classification of Functioning, Disability and Health (ICF;
WHO, 2001) is a way to describe and classify health and health-related domains (Figure 1). The focus expands from Body Structures and Functions to include the components Activity and Participation to describe how an individual takes part in society, and it is also used as a tool for looking at functional aspects of health. There is also a derived version of the ICF, the International Classification of Functioning, Disability and Health for Children and Youth (ICF-CY; WHO, 2001), designed to record characteristics of the developing child and the influence of the child’s environments. Within the context of the ICF, ‘participation’ refers to the nature and extent of an individual’s involvement in life situations.
Restrictions in participation represent the difficulties individuals can experience in life situations due to the circumstances of their health condition. The environmental factors are the physical, social and attitudinal environments in which people live and conduct their lives and are either barriers to or facilitators of a person’s functioning. Personal factors include race, gender and age and are not specifically coded in the ICF because of the wide variability across cultures. However, personal factors are included in the ICF framework (Figure 1), because although they are independent of the health condition they may influence how a person functions.
In clinical practice for children with CP, the use of the ICF structure can guide the choice of assessments and intervention. Decision makers in the field of childhood disability can use the ICF concept when developing policies and procedures for e.g. designing frameworks for care management tools that can be applied for health conditions such as CP. It would be desirable that research studies include the various interactive components of the ICF (Figure 1).
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Figure 1. Illustration of the interactions between the multidimensional components of the International Classification of Functioning, Disability and Health (ICF).
Environmental factors and personal factors must always be taken into consideration as they affect everything in an individual’s life (ICF; WHO, 2001).
1.3.1 Neuroanatomical correlates to speech and language impairment
The brain lesions of children with CP can be classified under the Body Structures component of the ICF. Little is known and understood about the neural bases of speech and language impairments in children, including children with CP. Many factors determine the consequences of a lesion of the developing brain, such as the age when it occurred, the site, the size, and whether the lesion is unilateral or bilateral. Several methods are used for the evaluation of the brain structures and activity in children with CP, e.g.
magnetic resonance imaging (MRI). MRI, and/or computed tomography (CT), is performed in about 90% of the children with CP in western Sweden (K. Himmelmann, personal communication, n.d., 2015). MRI findings in CP can be classified by timing of injury during gestation, which has provided a timetable for a structure-function relationship in CP (Krägeloh-Mann, 2004).
The neuroanatomical correlate of the different types of CP and impairments can be studied and related to communicative ability and speech ability (Himmelmann, Lindh, & Hidecker, 2013). In a study of 68 children with CP (Himmelmann, Lindh, & Hidecker, 2013), periventricular lesions were associated with more functional and effective speech and communicative ability levels classified with the Communication Function Classification System (CFCS; Hidecker et al., 2011). Cortical/subcortical and basal ganglia lesions were, on the other hand, significantly associated with the absence of
Health condition Cerebral Palsy
Body Structures &
Functions Activities Participation
Environmental Factors Personal factors
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speech and with less functional CFCS levels. Thus, the timing of the brain lesions seems important for communicative ability.
Liégeois, Mayes and Morgan (2014) reviewed recent studies on neuroimaging findings in children with speech and language impairment (without CP). Structural and functional anomalies in the left supramarginal gyrus were found, suggesting a possible deficit in sensory feedback or integration. In children with language impairment (also without CP), cortical and subcortical anomalies were reported in a widespread language network, but with little consistency across studies. The review highlighted the variability in neuroimaging technique in the studies as well as the heterogeneity within and across participants (Liégeois, Mayes, & Morgan, 2014). Only large-scale longitudinal studies of well-defined clinical subtypes will lead to a clearer picture of the neural bases of speech and language impairment in children, with or without CP.
1.3.2 Speech impairment
Descriptions of speech in children with CP can be classified under the Body Function component of the ICF. The motor speech impairment dysarthria is defined by Yorkston, Beukelman, Strand and Hakel (2010) as ‘a neurologic motor speech impairment that is characterised by slow, weak, imprecise or uncoordinated movements of the speech musculature’ (p.4). The motor impairments in children with CP may lead to dysarthria and the severity can vary from mild to severe involvement with inability to coordinate the respiratory, laryngeal, phonation, velopharyngeal, resonance and articulation subsystem. In general, reports on the occurrence of speech impairment in children with CP are scarce and the descriptions of speech ability differ considerably across studies. However, in four large-scale studies from Norway, Sweden, Iceland and Western Australia, impaired speech was reported in around 20% of children with CP (Andersen et al., 2008;
Himmelmann, Hagberg, Wiklund, Eek, & Uvebrant, 2007; Sigurdardottir &
Vik, 2011, Watson, Blair, & Stanley, 2006). In a recent population-based sample of children (4–6 years) with CP, 71 of 79 (90%) children had motor speech impairment (including children with severe speech impairment and without speech) (Mei, Reilly, Reddihough, Mensah, & Morgan, 2014).
Detailed descriptions of speech production in children with CP are rare. In an early study by Byrne (1959), it was shown that consonants in the medial and final positions were the most difficult. Workinger and Kent (1991) explored the speech characteristics of 18 children with CP (mean age 12;3 years) – nine with spastic CP, nine with dyskinetic CP. All children had severe gross motor impairment, but only one of them had cognitive impairment.
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Consistent hypernasality, breathy voice and change of voice quality were the most common auditory perceptual speech characteristics for the children with spastic CP. For the children with dyskinetic CP, reduced stress, inappropriate voice stoppage/release and slow rate were most common. They also had more severe articulation problems. Similarities between the speech profiles of the spastic and dyskinetic children were also found in terms of phonation, such as strained voice quality and harsh voice.
Children with CP and speech impairment with a developing speech sound system may have even more problems than adults with consonants of high phonetic complexity, as both developmental factors and the speech motor impairment may affect production of these speech sounds (Kim, Martin, Hasegawa-Johnson, & Perlman, 2010; Platt, Andrews & Howie, 1980).
Research on individuals with CP has mainly focused on adults and shows that high-complexity consonants, such as fricatives, requiring refined speech motor control, are more commonly misarticulated than speech sounds with lower complexity. Interestingly, Kim, Martin, Hasegawa-Johnson and Perlman (2010) showed that the speakers with CP with low speech intelligibility had more difficulties producing consonants of high complexity than the speakers with high intelligibility. The impact of phonetic complexity of words on intelligibility in children with CP has been explored by Allison and Hustad (2014). Sixteen children with CP (mean age 5 years, with and without motor speech impairment) participted in the study. Phonetic complexity was calculated using the consonant classification system described by Kim and colleagues (2010) in their study on speech in adults with CP. Consonants were assigned into different levels of articulatory complexity and each consonant had a phonetic complexity level value. The 119 naïve listeners made orthographic transcriptions of the children’s sentence productions. It was shown that phonetic complexity affected the intelligibility of the children with CP and speech impairment with variation observed across individual children. The results of their study suggest that reducing the phonetic complexity of utterances may aid in enhancing intelligibility in children with CP and speech impairment (Allison & Hustad, 2014). This shows that more knowledge on consonant production and phonetic complexity in speaking children with CP is needed.
1.3.3 Language impairment
How a child with CP produces and understands language can be classified under the Body Functions component of the ICF. There has been little research on language impairment in children with CP, but some information
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can be found in the few studies on how speech impairments in children with CP influence the use and development of language. Smith, Dahlgren Sandberg and Larsson (2009) found that children with CP (aged 5–13 years) without speech had significantly lower results than matched speaking controls on receptive grammar measured with TROG-2 (Bishop, 2003).
Bishop, Brown and Robson (1990) showed that children with CP (aged 10–
18 years) without speech and with severe motor speech impairment performed worse than typically developed (TD) children on a receptive vocabulary test, but performed equally well when receptive grammar was tested.
Receptive language comprehension is also required for story retelling (Dodwell & Bavin, 2008). A child’s ability to understand and produce narratives relies on a complex interaction between several language and social abilities (Norbury & Bishop, 2003) and memory (Dodwell & Bavin, 2008). Only one study is found on narrative ability in speaking children with CP (Holck, Dahlgren Sandberg, & Nettelbladt, 2011). Story retelling ability was examined with the Bus Story Test (BST; Renfrew, 1997; Svensson &
Tuominen-Eriksson, 2002) in ten Swedish-speaking children with CP without cognitive deficits (mean age 7;11 years). They were matched for age and gender with ten TD controls. There were no significant differences in scores of information or sentence length compared with the TD children. However, the grammar of the children with CP was less complex, with fewer subordinate clauses.
In this thesis, the short-term memory is the capacity of holding a small amount of information available in mind for a short period of time and working memory is the system that actively holds and manipulate multiple pieces of information in mind. The limited duration of short-term memory suggests that its contents spontaneously decay over time. The decay assumption is part of many theories of short-term memory, and the most notable may be Baddeley’s model of working memory (Baddeley and Hitch, 1974; Baddeley, 1986). Auditory presented material is registered into the short-term phonological store of the articulatory loop and visually presented information may gain access to the articulatory loop via a subvocal articulatory translation and rehearsal process (e.g. Salame and Baddeley, 1982).
Baddeley, Thomson and Buchanan (1975) have suggested that articulation rehearsal rate decides the amount of verbal material that can be maintained in memory. Interestingly, White, Craft, Hale and Park (1994) demonstrated that normal speech rates did not play a crucial role in determining the capacity of memory span. Eleven children with spastic diplegic CP without cognitive
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deficits, 5–11 years of age, and with subtle impairments in articulatory rate were compared with typically speaking controls. The children with CP did not show any memory span impairment compared with the children without speech rate deficits when tested with a memory span task that consisted of verbal repetition of one-, two- or three-syllable words (nouns). Quite different results have been reported for children with CP without speech.
Fifteen children in a Swedish study (5–12 years without speech) were matched with typically speaking controls (Larsson & Dahlgren Sandberg, 2008). These children with CP had problems with most memory functions, which was seen as a support for the hypothesis that impaired speech ability affects the children’s subvocal articulatory rehearsal (see e.g. Salame &
Baddeley, 1982) and therefore causes them memory problems.
Theory of mind (ToM) is another important ability when retelling a story. It i s defined as ‘the complex ability to explain and predict people’s behaviour wi th reference to mental states’ (p.1, Slaughter & Repacholi, 2003). For a functi onal ToM, it is considered that language and cognitive abilities interact (Mill er, 2004). In a study by Holck, Dahlgren Sandberg and Nettelbladt (2010), te n children with CP and intelligible speech (including two children with minor phonology and grammar impairment) had significantly worse results on ToM than matched TD children. Dahlgren, Dahlgren Sandberg and Larsson (2010) examined ToM in 16 children (mental age 4–9.5 years) with CP with such se vere speech impairment that people outside their families did not understand t hem. They performed significantly worse on ToM than the matched comparis on group of TD children. The low ToM results were discussed in terms of po or language abilities. Thus, the ability to predict and explain other people’s ac tions, the ToM ability, has accordingly been proven to be difficult, both for c hildren with CP with severe speech impairment (Dahlgren, Dahlgren Sandber g, & Larsson, 2010) and for children with CP and intelligible speech in the st udy by Holck, Dahlgren Sandberg, and Nettelbladt (2010).
1.3.4 Speech and language assessment
The majority of methods designed for assessing children with CP and speech and/or language impairment can be classified under the Body Function component of the ICF. Assessments of speech and language impairment can include both observations of spontaneous speech and language behaviour.
Standardised tests of speech and/or language abilities are used to determinate the severity of the impairment, and the possible direction for intervention (Hansson & Nettelbladt, 2007). Sometimes standardised tests have to be adapted to accommodate the difficulties of the children with CP, for example when pointing to pictures.
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Speech assessment of a child with CP often includes an overall rating of the severity of speech impairment. However, speech impairment in children with CP appears to involve all speech subsystems to some extent (e.g. Hustad, 2010) and consequently respiratory function, velopharyngeal function, phonatory function and articulation should be considered.
Regarding respiration, it is important to obtain information about any deficits affecting the speech production. The child’s usual speech production can give some indications of speech breathing problems. According to Hardy (1983), such indications are use of short utterances with inhalations between utterances, use of considerable generalised effort for speech production, slow speech and the sound of being ‘strained’ at the end of phrases. Hardy (1983) also suggested various types of speech activities that may be used to assess the respiratory function of a child with CP including various types of valving of the airstream while speaking. For example, the speaker may count as long as possible on one breath and also produce voiced and voiceless consonants in order to examine the respiratory subsystem.
Enough information about deviant velopharyngeal function can usually be provided via auditory-perceptual ratings of hypernasality and audible nasal air escape (Hardy, 1983). However, the interdynamics of the entire speech physiology process must be considered and, for example, deviations of the velopharyngeal function may be present due to dysfunction of one or more of the other speech subsystems. Finally, Hardy (1983) recommends careful assessment of articulation using narrow phonetic transcription in order to detect misarticulations as precisely as possible. The importance of examining the articulation was shown by Lee, Hustad and Weismer (2014) when investigating speech acoustic characteristics in 22 children with CP (mean age 5;7 years). The articulatory subsystem was the most independent contribution to speech intelligibility when a multiple speech subsystems approach was used.
Speech in children with CP can be assessed using spontaneous speech, repetition, naming or reading single words or sentences (e.g. Kent, 1996). In Sweden, the articulation and nasality test, SVANTE (Lohmander et al., 2014) is available with measures on articulation skill (percent correct oral consonants), articulation errors and nasality. Phonetic transcription of consonants and ordinal scale rating of nasality can be performed on single words, sentences and connected speech. The obtained results can be compared with reference data from children with typical speech development.
Language assessment made by the SLP of a child with CP can include tests
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of phonology, grammar, vocabulary (expressive and/or receptive) and receptive language comprehension. A variety of elicitation strategies are available, including imitation, naming, sentence supplementing, example sentences, retelling and spontaneous dialogues (Hansson & Nettelbladt, 2007). Expressive language can be tested with the ‘LuMat’ [Nya Lundamaterialet] (Holmberg & Stenkvist, 1983) or the Swedish test of grammar [GRAMmatiktest för Barn, GRAMBA] (Hansson & Nettelbladt, 2004), and phonology with the ‘Phoneme Test’ [Fonemtest] (Hellquist, 2013) or SVANTE (Lohmander et al., 2014) for consonant inventory and processes with normative data for preschool age children. Assessment of language comprehension is also important. Receptive grammar is commonly tested with the Test for Reception of Grammar Version 2 (TROG-2; Bishop, 2003) Receptive vocabulary can be assessed with the Peabody Picture Vocabulary Test (PPVT IV; Dunn & Dunn, 2007). Both receptive and expressive language can be evaluated with the Clinical Evaluation of Language Fundamental (CELF; Seme,Wiig, & Secord, 2004). Within clinical practice, all the above described instruments are used on a regular basis in order to assess children’s language ability. Narrative abilities have been found to be related with literacy ability, a valid and important predictor of longitudinal language abilities (Botting, 2002). Therefore a retelling test instrument such as the Bus Story Test (BST; Renfrew, 1997; Svensson & Tuominen-Eriksson, 2002) can be appropriate to use for children from the age of 4 years.
1.3.5 Communicative activity and participation assessment
Communicative participation is defined by Eadie et al. (2006) as ‘taking part in life situations where knowledge, information, ideas, or feelings are exchanged’ (p.309). The social and cultural environment is important to consider, as we know that other people's attitudes, values and beliefs affect a child's participation in daily activities (Law et al., 1999). A link between activity and social participation restrictions and severe communication impairment has been demonstrated in research on individuals with anarthria with complex communicative needs (Thirumanickam, Raghavendra, &
Olsson, 2011, Clarke et al., 2012; McFadd & Hustad, 2013). The importance of communication in order to facilitate participation in daily life activities is well known (Hidecker, 2010; Wilcox & Woods, 2011). However, there is limited knowledge about the activities and participation of children with CP and communication impairments.
Classification systems have been developed for individuals with motor speech impairment and communication functioning at an activity and participation level. Examples include the Viking Speech Scale (VSS;
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Pennington, et al., 2013) and the Communication Function Classification System (CFCS; Hidecker et al., 2011). The CFCS and the VSS can be used by a wide number of users including allied health professionals such as SLPs.
The VSS is used as a tool to classify the speech performance of children with CP (Pennington et al., 2013). Effectiveness of communication can be classified with the CFCS in order to determine the functional or daily impact of communication impairment (Hidecker et al., 2011). In a study by Mei, Reilly, Reddihough, Mensah and Morgan (2014), motor speech ability was classified in a community cohort of 79 children with CP (mean age 5;4 years) by the examiner and the parents using the VSS (Pennington, et al., 2013).
Communicative abilities at an activity and participation level were also classified using the CFCS (Hidecker et al., 2011) and the Functional Communication Classification System (FCCS; Barty & Caynes, 2009). Both the CFCS and the FCCS have five levels and are designed specifically for children with CP. The FCCS includes classification of both the familiarity of the communication partner and settings, whereas the CFCS classifies communication merely according to how effectively an individual sends and receives messages with familiar and unfamiliar communication partners. The VSS was used to estimate the occurrence of motor speech impairment in the cohort. Ninety percent of the children with CP had some degree of motor speech impairment according to the VSS. Moreover, it was found that activity and participation limitations increased with greater reductions in motor speech ability. The children in the study with mild motor speech impairment (VSS level II) displayed participation comparable to children without motor speech impairment. Participation was measured only based on parent report, i.e. the opinions of the children with motor speech impairment themselves were not included in this study. The authors therefore emphasised that the children’s own opinions about their communicative participation in various activities would have been a preferable complement (Mei, Reilly, Reddihough, Mensah, & Morgan, 2014).
1.3.6 Own and environmental opinions about communicative ability
Children with CP and speech impairment should be able to participate and communicate on equal terms with others in their family life and participate in all types of activities (UN, 1989).
Little is known about children’s own opinions about their speech and communicative ability, especially in children with CP. Interviewed preschool children with speech impairment (without CP) and their communication partners reported frustrating speech and listener problems (McCormack, McLeod, McAllister, & Harrison, 2010). The strategies they used to reach
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solutions, such as participating in speech intervention with SLPs, and also their use of informal strategies to address the listeners’ problem were described. One reason for the lack of information on how children with CP feel about their own speech and communicative ability is that there are no formal standardised tests. The Communication Attitude Test (CAT; Brutten
& Dunham, 1989), originally developed for children who stutter, has been used for this purpose. There is also a Swedish version of this test, CAT-S (Johannisson et al., 2009). It has been shown that children who stutter and children with voice and speech impairment relatedto cleft palate have more negative attitudes towards their own communication than children without these impairments (De Nil & Brutten, 1990; Havstam, Dahlgren Sandberg, &
Lohmander, 2011). Children with articulation problems, however, had less negative attitudes towards their speech (De Nil & Brutten, 1990). The children with CP themselves have the expert knowledge of the impact that speech impairment has on activities and participation in their lives, and therefore more such data from them would be desired.
Data in the area of children’s quality of life (QoL) has historically been taken from parent proxy reports. However, the World Health Organization (WHO, 1993) has recommended that measures of children’s QoL be obtained using subjective self-reporting whenever possible. Research involving healthy children has reported that parents generally proxy-report higher QoL than the children themselves (e.g. Theunissen et al., 1998), whereas parents of children with chronic conditions, such as epilepsy, proxy-report lower QoL than the children themselves (e.g. Ronen, Streiner, & Rosenbaum, 2003).
This is in line with findings from the European Study of Participation of Children with Cerebral Palsy Living in Europé (SPARCLE), where the children with CP self-reported significantly higher than the parents concerning participation and quality of life (Dickinson et al., 2007). Children with CP and speech impairment communicate with adult persons, such as their parents and their teachers, who are important communication partners for them in different ways and in different settings. The parents communicate mostly with them at home and the teachers in learning situations at school.
One way to document communicative abilities is to use questionnaires completed by an adult who knows the child well. Children’s Communication Checklist-2 (CCC-2; Bishop, 2003) is an instrument for this purpose. CCC-2 is a tool for identifying children with specific and pragmatic language impairment, and can also distinguish children with communication impairments and social interaction deficits who are in need of autism neuropsychiatric assessment. Mei et al. (2015) interviewed parents about their views of the activities and participation of their children with CP (4–9 years) with and without communicative impairments. The content of the
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interviews with the parents were mapped to the domains of the ICF-CY (WHO, 2001). They were primarily mapped to the domains of learning and applying knowledge, communication, mobility and interpersonal interaction.
The parents highlighted the potential negative impact of communication limitations on the activities and participation of their children, especially in the areas of relationships and independence. The opinions of the children themselves, the parents, the teachers and other professionals about the children’s communicative ability are important information in order to be aware of any discrepant opinions. This information might provide important knowledge in order to provide support in identified problem areas.
Furthermore, it may be a base for mutual discussions about what kind of communicative intervention would be best for the individual child
1.4 Rationale
More than half of children with CP have accompanying impairments, such as communication disabilities (Himmelmann, Beckung, Hagberg, & Uvebrant, 2006; Himmelmann & Uvebrant, 2011), that may be more disturbing than the actual motor impairment. Children with CP and speech impairment are a neglected group (Pennington,1999; Hustad, 2010; Egefors, 2012). According to the United Nations Convention on the Rights of the Child (1989), it is a child’s right to be able to communicate, children with CP and speech impairment included. It is therefore important to get an overview of the occurrence and distribution of speech, language and communicative disabilities and also explore both speech and language abilities thoroughly.
More understanding on these issues will increase the chances of finding appropriate intervention options for communicative improvements and participation in society. In addition, we do not know much about what the children themselves, or the important adults around them, think about their ability to communicate. There is therefore a need to learn more about this. In alignment with the need to explore speech, language and communicative ability in children with CP, the overall and specific aims outlined in Section 2 below have been chosen.
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2 AIM
The overall aim of this thesis was to explore and describe the speech and language ability of school-aged children with CP and speech impairment. The aim was also to investigate, describe and compare the communicative ability of the children from different perspectives.
The specific aim of each study was:
Study I: to describe and explore speech ability in a population-based study of children with CP (born 1999–2002) in relation to CP subtype, motor function, cognitive level and neuroimaging findings.
Study II: to investigate some speech characteristics of children with CP and speech impairment and study the relation between speech production, gross motor function and non-verbal cognitive level.
Study III: to explore the retelling ability of children with CP and speech impairment and the impact of expressive and receptive language, narrative discourse dimensions, auditory and visual memory, theory of mind and non- verbal cognition.
Study IV: to describe and compare communicative ability in school-aged children with CP and speech impairment from the perspectives of the children themselves, their parents, their teachers and their SLP.
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3 MATERIAL AND METHODS
3.1 Participants
The participants in Study I were 129 children (66 girls, 63 boys) with CP from a population-based cohort born in 1999–2002 in the county of Västra Götaland, which is part of the Panorama of CP in western Sweden study area (Himmelmann, Hagberg, & Uvebrant, 2010). The child’s local paediatric neurologist diagnosed all participating children with CP when they were between 4 and 8 years old. CP was defined according to Rosenbaum et al.
(2007) as a group of disorders of the development of movement and posture that are attributed to non-progressive disturbances in the foetal or infant brain. A flow chart (Figure 2) shows the number of children included in each of the four studies in the thesis.
Figure 2. Number of children participating in each of the four studies.
Study I
129 children with CP: 61 without impaired speech, 27 with speech impairment, 41 non-verbal
Speech, language and communication assessment of the children with speech impairment: n=22 (five declined)
Study II - Speech
19 children with with speech impairment (three excluded)
Study III - Language
15 children with speech impairment (seven excluded)
Study IV - Communication
16 children with speech impairment (six excluded)
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The 27 children (14 girls, 13 boys) ages 9;2 to 12;10 (mean 11;2 years ) with speech impairment, i.e. 21% of the 129 children, were invited to participate in an in-depth assessment of speech, language and communication. Twenty- two of them accepted the invitation and five declined. Table 1 gives an overview of the individual children participating in the studies in this thesis.
It also provides background data, earlier reported by Himmelmann, Hagberg and Uvebrant (2010) and Himmelmann and Uvebrant (2011), on type of CP, level of gross motor function and neuroimaging findings.
In Study II, dealing with speech characteristics in children with CP, three of the 22 children from the population-based study were excluded. One child was not able to take part in the assessment due to epilepsy, one child did not produce enough speech during the assessment and one child could not participate due to visual impairment as the test included picture naming.
Thus, 19 Swedish-speaking children (9 girls, 10 boys) age 9;2 to 12;9 (mean 11;2 years) participated.
In Study III, concerning the impact of language abilities on retelling ability, seven of the 22 children from the population-based study were excluded (Table 1). One child was not able to take part in the assessment due to epilepsy, five children did not use the five utterances required for the assessment of retelling, and one child did not participate due to visual impairment. The study group finally comprised 15 Swedish-speaking children (7 girls, 8 boys), age 9;2 to 12;9 (mean 11;0 years).
In Study IV, about different perspectives on communicative ability, 16 children (7 girls, 9 boys) age 9;2 to 12;10 (mean11; 2 years) met the criteria of speaking several sentences and participated (Table 1). Six of the 22 children from the population-based study were excluded (Table 1). One child was not able to take part in the assessment due to epilepsy, one child had severe cognitive impairment, the parents of one child did not speak/read Swedish and three children did not speak in several sentences, which was required for the assessment. The children themselves, their parents and teachers participated.
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Table 1. Description of the participants in Study II, Study III, and IV.
Study II n=19/22
Study III n=15/22
Study IV n=16/22
Gender Age Type of CP
GMFCSa ** VSSb
S1 Child A Participant 1
F 9;6 USCP I II
S2 Child B Participant 2
M 9;9 USCP I II
S3* Child C* Participant 3*
M 12;8 USCP I II
S4 Child D Participant 4
M 11;11 USCP I II
S5 Child E Participant 5
F 12;9 USCP I II
S6 Child F Participant 6
F 12;8 USCP I II
S7 Child G Participant 7
F 9;8 BSCP IV II
S8 Child H Participant 8
M 10;7 BSCP IV II
S9 Child I Participant 9
M 11;3 USCP III II
S10* Child J* X M 11;8 USCP I II
S11 X X M 11;8 BSCP I III
X X Participant
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M 12;10 BSCP IV II
S12* X X F 12;8 BSCP IV III
S13 X X F 11;9 BSCP III III
D1 Child K Participant 11
M 11;10 Dys-
kinetic
III II
D2 Child L Participant 12
F 9;2 Dys-
kinetic
II II
A1 Child M Participant 13
F 10;1 Ataxic I II
A2 Child N Participant 14
F 9;2 Ataxic IV III
A3 X Participant
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M 10;7 Ataxic I III
A4 Child O Participant
16 M 12;1 Ataxic I II
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Notes (Table 1): X not part of this study; F, Female; M, Male * Bilingual children** Himmelmann et al.
(2010). aGMFCS, Gross Motor Function System has five levels for describing severity of gross motor impairment with level I — walks without limitations; level II — walks with limitations; level III — walks with adaptive equipment assistance; level IV — self-mobility is limited, likely to be transported in a wheelchair; level V — transported in a wheelchair. bThe Viking speech scale has four levels: level I – speech is not affected by motor disorder; level II – speech is imprecise but usually understandable to unfamiliar listeners; level III – speech is unclear and not usually understandable to unfamiliar listeners;
level IV – no understandable speech.
3.2 Ethical consideration
All studies were approved by the Regional Ethical Review Board in Gothenburg (Dnr: 145-07; Dnr: 639-10). The parents of the children participating in the studies received oral and written information before consent was obtained. The children themselves were given information about the study before they agreed to participate.
3.3 Procedures
In Study I, all available information about the population-based cohort of 129 children with CP wase reviewed, i.e. the medical records and information from SLPs working with the child concerning the speech ability of the participants. The data collection was conducted by an SLP (the author). In Studies II, III and IV, the same SLP met all children individually. The assessments took place in a separate room at the children’s schools, except for one child, who was assessed at home. Audio and video recordings of the speech and language testing were performed in a standardised manner using high-quality equipment. In Study IV, data were collected via questionnaires directed to parents and teachers as well as questions/statements given to the children themselves.
3.3.1 Test instruments
The participants of the studies (n=22) took part in in-depth and broad assessment of speech, language and communication skills, and the following standardised test instruments were used:
• The Swedish Articulation and Nasality TEst (SVANTE;
Lohmander et al., 2005) was used for assessment of consonant production and nasality. The SVANTE test includes pictures designed to elicit one- and two-syllable single words with target oral consonants (59), nasal
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consonants (5) and s-clusters (10). The SVANTE test also includes the repetition of sentences and the elicitation of connected speech, but in this study the 59- single-word sample was used for assessment of the oral consonant production. The following consonants were assessed: /p, /t/, /k/, /b/, /d/, /ɡ /, /f/, /s/ and /ɕ/. The speech sample thus includes all Swedish oral stops, the two sibilants and one labiodental voiceless fricative, all considered vulnerable to articulation impairments, For each consonant included there are seven possible realisations (three in initial position, two in medial and two in final position), except for /ɕ/ with three possible realisations in initial position.
• The Bus Story Test (BST; Renfrew 1997, Swedish version; Svensson and Tuominen-Eriksson, 2002) was used for the assessment of oral retelling ability. BST is a picture-based test instrument for the ages of 3;9 to 8;5 years. BST includes 12 cartoon pictures and no written words. The participants were told the story orally (by the author) while looking at each picture and then asked to retell the story as closely to the original as possible using the pictures as prompts.
• The Test for Reception of Grammar (TROG-2; Bishop, 2003) was used for the assessment of receptive grammar at sentence level. Receptive vocabulary was assessed with the norm-referenced instrument Peabody Picture Vocabulary Test (PPVT-IV; Dunn & Dunn, 2007) including 228 picture-based test items.
• Non-verbal cognitive level was tested with the Raven’s progressive matrices (RCPM; Raven, Court, & Raven, 1986).
• The Corsi block-tapping task (CB) (Milner, 1971) was used to measure visuo-spatial memory. The examiner pointed to a number of blocks in a certain order and the participant was asked to point to the same blocks in their order of presentation.The forward version was a
measure of visuo-spatial short-term memory (STM) and the backward for visuo-spatial working memory (WM).
